open access

Vol 50, No 2 (2019)
Case Report / Kazuistyka
Published online: 2019-08-23
Submitted: 2019-04-23
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Progressive arthropathy in the course of immune tolerance induction failure in a child with hemophilia A and FVIII inhibitor – a case report

Paweł Łaguna, Anna Klukowska, Jarosław Ćwikła, Michał Matysiak
DOI: 10.2478/ahp-2019-0013
·
Acta Haematol Pol 2019;50(2):81-84.

open access

Vol 50, No 2 (2019)
Case Report / Kazuistyka
Published online: 2019-08-23
Submitted: 2019-04-23

Abstract

We present the case of an 18.5-year-old boy with hemophilia A and FVIII inhibitor detected at the age of 8 months. No persistent inhibitor eradication was achieved, despite three attempts of immune tolerance induction (ITI) with temporary inhibitor resolution and subsequent administration of 11 doses of rituximab. In spite of hemostatic treatment, rehabilitation, and radiosynovectomy, the patient suffered from recurrent bleeding episodes into almost all major joints, which resulted in the development of progressive hemophilic arthropathy. Currently, due to high frequency of bleeding episodes, the patient is being treated prophylactically and receives prophylaxis with activated prothrombin complex concentrate (aPCC).

Abstract

We present the case of an 18.5-year-old boy with hemophilia A and FVIII inhibitor detected at the age of 8 months. No persistent inhibitor eradication was achieved, despite three attempts of immune tolerance induction (ITI) with temporary inhibitor resolution and subsequent administration of 11 doses of rituximab. In spite of hemostatic treatment, rehabilitation, and radiosynovectomy, the patient suffered from recurrent bleeding episodes into almost all major joints, which resulted in the development of progressive hemophilic arthropathy. Currently, due to high frequency of bleeding episodes, the patient is being treated prophylactically and receives prophylaxis with activated prothrombin complex concentrate (aPCC).

Get Citation

Keywords

hemophilia A; factor VIII inhibitor; ITI; bypassing agents

About this article
Title

Progressive arthropathy in the course of immune tolerance induction failure in a child with hemophilia A and FVIII inhibitor – a case report

Journal

Acta Haematologica Polonica

Issue

Vol 50, No 2 (2019)

Pages

81-84

Published online

2019-08-23

DOI

10.2478/ahp-2019-0013

Bibliographic record

Acta Haematol Pol 2019;50(2):81-84.

Keywords

hemophilia A
factor VIII inhibitor
ITI
bypassing agents

Authors

Paweł Łaguna
Anna Klukowska
Jarosław Ćwikła
Michał Matysiak

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